Only four cases, including the one presented here, have been confirmed with brain biopsy [6,8,10]. between the first and thirteenth days since symptom onset, and the CSF profile was not typical for HSV encephalitis. The patient underwent a brain biopsy, which confirmed the presence of HSV. She continued to worsen despite aggressive seizure control and six days of empiric acyclovir. Unfortunately, she expired despite the reinitiation of acyclovir. When faced with the classical features of encephalitis in the immunocompromised, the suspicion of HSV should remain high despite negative PCR results. The completion of a full course of acyclovir in the absence of clinical improvement should be considered. strong class=”kwd-title” Keywords: case-based review, review, Naphthoquine phosphate case report, encephalitis, vasculitis, hsv pcr, status epilepticus Introduction Polymerase chain reaction (PCR) detection of viruses and bacteria has revolutionized diagnostic approaches to infectious diseases. Improvements in technology have made PCR testing a practical and efficient approach to the recognition and management of many life-threatening infections?such as herpes simplex virus (HSV) encephalitis [1]. This is in part due to its impressive specificity, cited as being between 95% and 99% with sensitivity between 94% and 98% [2]. However, limitations to these technologies remain and over the past 20 years, several instances of PCR-negative HSV encephalitis have been documented [3-10], raising important questions on how to approach testing such as the timing of testing in relation to symptom onset, need for repeated lumbar punctures, and alternative confirmatory methods. This is especially true in immunocompromised individuals, who are at risk for many variants and obscure entities that may not be detected by standard screening measures. This is of particular concern, as advances in treating autoimmune disorders, organ transplantation, and immunotherapies for cancers have significantly increased the number of patients Naphthoquine phosphate who are on chronic immunosuppression. We present the case of a 62-year-old woman with a past medical history significant for systemic lupus erythematosus (SLE) and p-ANCA vasculitis (on immunosuppression) who was found to have PCR-negative HSV encephalitis. We also present a review of all identifiable reports of PCR-negative HSV encephalitis in the past 20 years. To our knowledge, this is the first case of PCR-negative HSV encephalitis in a patient with p-ANCA vasculitis. PAX3 Case presentation The patient was in her usual state of health when she became febrile to?101F (38.3 C).? The following day, she developed confusion with a leftward head version and leftward gaze deviation. She presented to an outside hospital, where she had multiple?episodes?of?witnessed events concerning for focal motor seizures with progression to generalized bilateral tonic-clonic activity. She was determined to be in status epilepticus and was treated?with levetiracetam. Due to concern for meningitis, her initial regimen included methylprednisolone and empiric? antibiotic and antiviral coverage with vancomycin, ampicillin, ceftriaxone, acyclovir, and sulfamethoxazole/trimethoprim. Ampicillin was started Naphthoquine phosphate by the community hospital for empiric coverage of listeria meningitis given the patients age and immunocompromised status. Her serum creatinine upon presentation to the community hospital was 1.1 mg/dL, which was slightly worse than her known baseline of 1 1.04 mg/dl from two weeks prior. She was switched from ampicillin to sulfamethoxazole/trimethoprim to avoid further nephrotoxicity. Routine EEG showed diffuse moderate to severe slowing without epileptiform activity. MRI?brain demonstrated nonspecific restricted diffusion in the right basal ganglia and frontal and temporal?lobes (Figure ?(Figure1A).1A). The pertinent laboratory results showed a plasma sodium level of 125 mmol/L and an absolute neutrophil count of 1 1.1 thous/mcL. Her baseline plasma sodium level was 140 mmol/L and absolute neutrophil count was 5.2 thous/mcL one month prior. Cerebrospinal fluid (CSF) analysis revealed an elevated white blood cell count to 13/mm3, a glucose concentration of 78 mg/dL, and a protein level of 41 mg/dL (Table ?(Table1).1). The patients blood cultures were negative. Based on available laboratory data, the initial clinical impression was status epilepticus in the setting of hyponatremia. Her neurological status did not improve with the normalization of plasma sodium levels. Table 1 Lumbar puncture resultsOSH: results obtained from the outside hospital; RBC: Naphthoquine phosphate red blood cells Date of Hospital CourseDay 1 (OSH)Day 5Day 13AppearanceNot reportedCLEARCLEAR/COLORLESSRBC CSF (mm3)Not reported1 (H)22 (H)?Nucleated Cells (mm3?)13 (H)1 ?1?CSF Lymphocytes %Not reported7733?Glucose, CSF ?(mg/dl)78 (H)97 (H)43?Total Protein, CSF (mg/dl)41 (H)49 (H)62 (H)Lactate, CSF (mmol/l)-3.5 (H)-Albumin, CSF (mg/dl)-20- Open in a separate window Figure 1 Open in a separate window Radiologic progression of disease seen on MRI brainA) Day 1: MRI brain FLAIR demonstrates right temporal and frontal lobe areas of hyperintensity and ADC/DWI mismatch consistent with HSV encephalitis and acute infarction (red arrows). B) Day 11: MRI brain FLAIR shows progression to bilateral involvement. C).
